Literature DB >> 32331917

Expanding the disease phenotype of ADSSL1-associated myopathy in non-Korean patients.

Magdalena Mroczek1, Hacer Durmus2, Sunita Bijarnia-Mahay3, Ana Töpf1, Roula Ghaoui4, Samantha Bryen5, Jennifer Duff1, Eleina England6, Sandra T Cooper7, Daniel G MacArthur6, Volker Straub8.   

Abstract

Adenylosuccinate synthase (ADSSL1) is a muscle specific enzyme involved in the purine nucleotide cycle and responsible for the conversion of inosine monophosphate to adenosine monophosphate. Since 2016, when mutations in the ADSSL1 gene were first described to be associated with an adult onset distal myopathy, nine patients with compound heterozygous variants in the ADSSL1 gene, all of Korean origin, have been identified. Here we report a novel ADSSL1 mutation and describe two sporadic cases of Turkish and Indian origin. Many of the clinical features of both patients and muscle histopathology and muscle MRI findings, were in accordance with previously reported findings in the adult onset distal myopathy individuals. However, one of our patients presented with progressive, proximally pronounced weakness, severe muscle atrophy and early contractures. Thus, mutations in ADSSL1 have to be considered in patients with both distal and proximal muscle weakness and across various ethnicities.
Copyright © 2020. Published by Elsevier B.V.

Entities:  

Keywords:  ADSSL1; Adenylosuccinate synthase; Adult onset distal myopathy; MPD5

Mesh:

Substances:

Year:  2020        PMID: 32331917     DOI: 10.1016/j.nmd.2020.02.006

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  2 in total

1.  Panorama of the distal myopathies.

Authors:  Marco Savarese; Jaakko Sarparanta; Anna Vihola; Per Harald Jonson; Mridul Johari; Salla Rusanen; Peter Hackman; Bjarne Udd
Journal:  Acta Myol       Date:  2020-12-01

2.  Improving the efficacy of exome sequencing at a quaternary care referral centre: novel mutations, clinical presentations and diagnostic challenges in rare neurogenetic diseases.

Authors:  Christopher Grunseich; Nathan Sarkar; Joyce Lu; Mallory Owen; Alice Schindler; Peter A Calabresi; Charlotte J Sumner; Ricardo H Roda; Vinay Chaudhry; Thomas E Lloyd; Thomas O Crawford; S H Subramony; Shin J Oh; Perry Richardson; Kurenai Tanji; Justin Y Kwan; Kenneth H Fischbeck; Ami Mankodi
Journal:  J Neurol Neurosurg Psychiatry       Date:  2021-06-08       Impact factor: 10.154

  2 in total

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